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Mullerian adenosarcoma of the cervix is a rare biphasic tumor composed of a benign epithelial component and a malignant stromal component. Here, we report a rare case of Mullerian adenosarcoma of the cervix in an adolescent girl treated with hysteroscopic resection. A 16-year-old girl presented to the Affiliated Hospital of North Sichuan Medical College in April 2017 with a 1-year history of a painless vaginal mass. Ten hours before presentation, she had experienced rapid enlargement of the vaginal mass and mild vaginal bleeding. Sixteen hours after hospitalization, a mass measuring 14 × 10 × 4 cm was spontaneously expelled from the vagina. Histopathological examination of the mass confirmed a diagnosis of Mullerian adenosarcoma. On May 10, 2017, the patient underwent hysteroscopic resection of a cervical lesion and partial cervical resection. After nearly 7 years of follow-up, the tumor has not recurred. Mullerian adenosarcoma of the cervix is difficult to diagnose in adolescents. For young women affected by low-risk early-stage Mullerian adenosarcoma of the cervix, fertility-preserving treatment using hysteroscopic resection with robust follow-up is a reasonable management option. [ABSTRACT FROM AUTHOR]

Adenosarcoma of the uterus with Sarcomatous Overgrowth (SO) and Heterologous Elements (HE) is a rare and aggressive neoplasm that shares common clinical and radiological presentations. It is also referred to as Müllerian adenosarcoma, a slow-growing neoplasm characterised by a classical biphasic pattern comprising benign epithelial and malignant mesenchymal tissue. Adenosarcoma with a high-grade stromal component has an increased risk of recurrence. Hereby the authors present a case of 39-year-old female who was nulliparous, reported to the Department of Obstetrics and Gynaecology with abdominal pain, menorrhagia, and burning micturition. Her medical history included diabetes, hypertension, hypothyroidism, and morbid obesity. She had a past history of similar complaints, during which an endometrial biopsy had shown hyperplasia without atypia. She was lost for follow-up and returned after one and a half years, having developed endometrial hyperplasia with atypia. She returned only after six months with similar but more severe complaints. Clinically and radiologically, there was a polypoidal mass protruding through the cervical canal. The hysterectomy specimen revealed a tumour with histopathological features of Müllerian adenosarcoma with SO. Interestingly, both ovaries were enlarged, exhibiting marked stromal hyperplasia. A panel of immunohistochemical markers, including CD10, Ki67, S-100, desmin, Estrogen Receptor (ER), and Progesterone Receptor (PR), was used, and the findings were unusual for an adenosarcoma with high-grade stromal histology. The chronological progression from endometrial hyperplasia without atypia to hyperplasia with atypia in a nulliparous woman, along with bilateral ovarian stromal hyperplasia, represents a classical pattern of events. [ABSTRACT FROM AUTHOR]

Background: Cervical mullerian adenosarcoma is a rare uterine sarcoma, especially in young women. Its pathological features are low-grade malignant tumors with bidirectional differentiation, and the degree of malignancy is similar to that of low-grade endometrial stromal sarcoma. This paper reports the case of a young asexual patient who has been closely followed up after tumor resection and has not had any recurrences. Case presentation: A 20-year-old, young asexual woman was diagnosed with cervical mullerian adenosarcoma with sarcomatous overgrowth (MASO). Cervical tumor resection was performed after admission, and the resection margin was negative. After the operation, she refused to undergo secondary surgery due to fertility requirements and did not receive adjuvant treatment. The patient was closely followed up after the operation and has not yet relapsed. Conclusion: A young woman with cervical MASO did not receive adjuvant treatment after cervical tumor resection. For women with fertility requirements, close follow-ups should be conducted after the operation to guard against tumor recurrence and radical tumor resection should be performed as early as possible after the patient no longer requires their fertility. [ABSTRACT FROM AUTHOR]

Mullerian adenosarcoma of the uterine cervix generally displays cervical polyps and can be often misdiagnosed as benign endocervical polyps both clinically and pathologically. A 52-year-old woman presented with a two-month history of postmenopausal bleeding. Pelvic examination revealed a large polyp-like mass protruding from the cervical ostium. Two preoperative consecutive biopsies showed a histologic diagnosis consistent with an endocervical polyp. Pelvic magnetic resonance imaging (MRI) depicted a lobulated well- enhancing endocervical mass, suggestive of an endocervical polyp based on the apparent diffusion coefficient (ADC) value. The patient was subjected to total hysterectomy with bilateral salpingectomy. Microscopically, the tumor was composed of epithelial and stromal components. The final pathologic diagnosis was Mullerian adenosarcoma of the cervix. The authors presented an unusual case that demonstrated an endocervical polyp on magnetic resonance imaging and two consecutive biopsies preoperatively. [ABSTRACT FROM AUTHOR]

Introduction: Uterine adenosarcoma mostly seen in elderly and there are limited data on appropriate approach in premenopausal patient and the accurate prognosis. Herein we aimed to present a young case of Müllerian adenosarcoma of uterus which had only 9 years old. Case report: A 9year old pre pubertal girl presented to the gynecology department of Isfahan University of Medical Sciences-Iran with the vaginal bleeding. An intrauterine mass was noted via hysteroscopic examination as the reason of abnormal bleeding. The final pathology revealed Müllerian adenosarcoma of uterus. Subsequently hysterectomy was done due to persistent perfused vaginal bleeding; with ovarian preservation. Conclusion: Due to rarity of Müllerian adenosarcoma, the gynecologists and pathologists should be reminded about the possibility of the diagnosis in young patients too. [ABSTRACT FROM AUTHOR]

Background: Adenosarcoma arises most commonly in the uterus. Rarely, it may arise from extrauterine sites, such as the pelvic peritoneum and rectovaginal septum. Two cases of extrauterine Müllerian adenosarcoma arising from pelvic endometriosis are discussed. Cases and Results:Case 1: A 63-year-old woman presented with vaginal bleeding and a rectovaginal nodule. She underwent an extended hysterectomy with resection of a rectovaginal mass. Histology revealed endometriotic tissue. She returned 21 months later with abdominal distension, and underwent resection of large pelvic abdominal masses. Histology showed an adenosarcoma with sarcomatous overgrowth. Her disease progressed despite chemotherapy, and she died 5 months later. Case 2: A 70-year-old woman with previous breast cancer presented with urinary retention. Computed tomography showed a 13-cm pelvic mass. She underwent total abdominal hysterectomy and salpingo-oophorectomy. Intraoperatively, dense pelvic endometriosis was seen. Histology showed a stage 1C borderline endometrioid adenofibromatous tumor. She also underwent resection of a 10-cm incidentally discovered pelvic mass 18 months later. Histology showed a low-grade adenosarcoma arising from pelvic endometriosis. She was started on letrozole, and remained well postoperatively. Conclusions: Compared to uterine adenosarcoma, an extrauterine location confers a higher mortality and recurrence risk. Higher stromal grade or sarcomatous overgrowth worsen the prognosis, whereas an association with endometriosis is protective. The mainstay of treatment is surgery. Some surgeons recommend fertility-sparing surgery when there is no sarcomatous overgrowth, while others advocate radical surgery with adjuvant chemotherapy or radiotherapy. Further research is needed to define screening, treatment, and follow-up better. Clinicians should consider this rare diagnosis in any woman presenting with a pelvic mass with a history of endometriosis. [ABSTRACT FROM AUTHOR]

Background Uterine adenosarcoma with sarcomatous overgrowth (ASSO) is a rare and aggressive disease. A case of a 16-year-old girl with uterine ASSO is reported herein. The patient received surgical resection and chemotherapy and remained alive without disease 11 months after the surgery. Case A 16-year-old girl was diagnosed with uterine ASSO, International Federation of Gynecology and Obstetrics (2009) stage I c. She underwent total abdominal hysterectomy, bilateral salpingectomy, and chemotherapy. She remains alive and there was no evidence of tumor recurrence on follow-up physical, laboratory, and ultrasound scan examinations. Summary and Conclusion Surgery is the primary treatment for uterine ASSO, total abdominal or laparoscopic-assisted vaginal hysterectomy with or without bilateral salpingo-oophorectomy are recommended. Early surgical resection might increase survival of uterine adenosarcoma. Long-term follow-up of the patients is recommended because of the high chance of recurrence. [ABSTRACT FROM AUTHOR]

Objective The aim of this study was to examine a series of clinically identified cervical polyps and determine the incidence of significant histologic and concurrent cytologic findings. Methods Consecutive cervical polyps from January 2000 through September 2012 were retrieved from the hospital laboratory information system. Histologic evaluation of these polyps was performed, followed by a chart review of clinical findings and correlation with the immediately prior or concurrently collected cervical Papanicolaou (Pap) test results, when available. Results A total of 369 cervical polyps were identified and reviewed. The patient ages ranged from 18 to 87 years (mean 46.5 years). Eight polyps demonstrated squamous dysplasia (6 Cervical Intraepithelial Neoplasia/CIN I, and 2 CIN II/III), while 6 had malignant or atypical/potentially malignant features (2 adenosarcoma, 2 atypical polyps concerning for Mullerian adenosarcoma, 1 endometrioid endometrial adenocarcinoma and 1 adenocarcinoma in-situ). An increased incidence of atypical squamous cells of undetermined significance (ASCUS) and atypical glandular cells not otherwise specified (AGC NOS) Pap diagnoses (12.7% and 6.1%, respectively) was found in women with benign polyps on biopsy. Discussion We demonstrated a higher rate of clinically significant histologic findings in cervical polyps (14 of 369 cases, 3.7%) compared to previously reported studies. The increase in ASCUS and AGC Pap results was most likely related to reactive and inflammatory changes present in benign polyps. Our results suggest that removal of all cervical polyps with subsequent histologic review is warranted. [ABSTRACT FROM AUTHOR]

Background Müllerian adenosarcomas of the cervix are composed of benign epithelial and malignant stromal components. The purpose of this report is to describe the clinical and histologic difficulties in diagnosis and to propose fertility-preserving management of low-grade lesions. Case A 14-year-old girl presented with a friable lesion found to originate from the anterior cervical lip. Initially, clinical suspicion was for sarcoma botryoides, however, pathologic evaluation revealed a low-grade cervical Müllerian adenosarcoma. Cold knife conization was performed, and the mass was resected with clear margins. Summary and Conclusion Müllerian adenosarcoma of the cervix is difficult to diagnose in adolescents because of features more commonly associated with alternative diagnoses. For patients with low-grade lesions desiring future fertility, local excision with close follow-up is reasonable. [ABSTRACT FROM AUTHOR]

Müllerian adenosarcomas most commonly arise in the uterine corpus from the eutopic endometrium. Occasionally, they arise in the cervix, vagina, broad and round ligaments, and ovaries and rarely in extragenital sites, usually in association with endometriosis. The authors report a rare case of extraendometrial, intramural adenosarcoma of low grade arising from a subserosal adenomyoma in a 46-year-old woman who presented with vaginal bleeding. No evidence of eutopic endometrial origin was identified; the adenosarcoma showed only limited myometrial invasion and no serosal involvement. Because of its subserosal location, the tumor would be overstaged as IC (deeply myoinvasive) in the new FIGO staging system. Stage IC tumors would normally be considered for adjuvant treatment, which would be inappropriate in this case. To avoid overstaging and inappropriate treatment, it is important to recognize that adenosarcoma can, rarely, arise in adenomyomas (or adenomyosis). [ABSTRACT FROM AUTHOR]

Non-puerperal uterine inversion due to uterine sarcomas represents a very rare event with no reliable estimate of frequency in the literature. Clinically, the diagnosis of inversion may be difficult, as far as imaging procedures are concerned, although ultrasonography may prove to be useful. However, some characteristics such as the indentation of the fundic area and a depressed longitudinal groove extending from the uterus to the center of the inverted portion are difficult to recognize. Moreover, there is no specific computed tomography feature accurate enough to aid in the differential diagnosis. Here, we report a case of uterine inversion due to Müllerian uterine adenosarcoma whose preoperative workup and diagnosis took advantage of the application of magnetic resonance imaging. Copyright © 2012 S. Karger AG, Basel [ABSTRACT FROM AUTHOR]

Rationale: There is little knowledge regarding mullerian adenosarcoma and its metastasizing pattern. Aim: Our objective was to evaluate the impact on the prognostic of the patient brought by tumor metastasizing to the retroperitoneum, to analyze the particularities of the treatment in such cases and to bring a significant change in the early therapeutic attitude to mullerian adenosarcomas. Methods and results: We present a first case report of a hypervascularized retroperitoneal metastasis from an initial low-grade uterine adenosarcoma. The presence of such a metastasis brought the worst prognostic factor to the patient. Conclusions: We consider that in front of a macroscopic polypoid mass there should be an active change in the diagnosis and in the therapeutic attitude. The frequent confusion between similar histopathological entities with different aggressiveness states and specific treatment responses, the poor outcome at advanced tumor stages, frequently in young patients, should trigger a universal remodeling in approaching these tumors. [ABSTRACT FROM AUTHOR]

: BackgroundPrimary adenosarcoma arising in vaginal endometriosis poses a diagnostic challenge, especially in superficial vaginal biopsies.: CaseA 56-year-old woman, with a prior diagnosis of ovarian endometriosis, presented with a rapidly enlarging mass of the vaginal vault. Two prior biopsies were benign and showed endometriosis. The third vaginal biopsy revealed benign endometriotic glands cuffed by a cellular stroma with moderate cytologic atypia, a histological appearance diagnostic of Mu¨llerian adenosarcoma. A 16-cm vaginal mass that had infiltrated the pelvic structures was resected.: ConclusionsClose clinical follow-up of extrauterine endometriosis and clinical–pathologic correlation is necessary. Histological features such as cellular stromal cuffing around benign endometriotic glands are critical in arriving at a timely diagnosis of adenosarcoma in patients with persistent extrauterine endometriosis, even in superficial vaginal biopsies. [Copyright &y& Elsevier]

Background: Uterine adenosarcoma, a rare uterine tumor subtype, is a biphasic tumor consisting of epithelial and mesenchymal elements. To date, there is no research comparing the histopathological features and immunohistochemistry of primary and recurrent tumors; furthermore, the relationship between pathology and the clinical course remains unclear. We reviewed the pathology and immunohistochemical features of patients with adenosarcoma and investigated the relevance of the histomorphological features to the clinical course. We also compared the immunohistochemical features of the primary and recurrent tumors. Methods: The data of seven patients with adenosarcoma who underwent surgery in our hospital were evaluated. We performed immunohistochemical staining for the progesterone receptor, estrogen receptor, p53, and two Switch/Sucrose Non-Fermentable chromatin remodeling proteins (SMARCA4, BCOR), which were recently developed for the undifferentiated sarcoma diagnosis in addition to conventional staining methods. Results: All patients had International Federation of Gynecology and Obstetrics stage IB–IC diseases. All tumors were polypoid and every patient presented with abnormal uterine bleeding. Six patients aged over 50 years and were menopausal; one patient aged under 50 years and was non-menopausal (average age: 59.1 years). Histologically, the sarcomatous components were homologous and heterogenous in six and one patient, respectively. Four and three cases were recurrent and non-recurrent, respectively. The recurrent patients showed high-grade morphology with sarcomatous overgrowth and were negative for ER and PR. Three recurrences could be evaluated by imaging, showing recurrence only in a distant area; biopsy specimens from these tissues revealed the identical mesenchymal component found in the primary tumor without a benign epithelial component. Immunohistochemical staining results were also similar to the corresponding of the original tumor, except for the p53 expression in one patient. At the primary site, p53 was overexpressed in two recurrent patients and had a wild-type level in one recurrent patient; however, all three recurrent tissues showed p53 overexpression. None of our patients showed SMARCA4 loss, and BCOR expression was positive in one case. Conclusions: Initial pathological adenosarcoma analysis with appropriate immunohistochemical staining is vital for prognostic assessment. p53 expression might increase at recurrence. SMARCA4 and BCOR might not be an index of malignancy. [ABSTRACT FROM AUTHOR]

Abstract: Background.: Primary vaginal adenosarcomas are extremely rare, and typical adenosarcomas are of low malignancy. However, aggressive forms with sarcomatous overgrowth have been reported, those appear to have a poor prognosis. Case.: A 52-year-old woman who had undergone prior surgery for uterine leiomyoma and an ovarian cyst (total abdominal hysterectomy and left salpingo-oophorectomy) presented 10 years later with a rapidly enlarging tumor arising from the vaginal cuff. Repetitive biopsy samples revealed a mixture of benign epithelial gland and malignant stromal components with periglandular stromal hypercellularity and sarcomatous overgrowth. A histological diagnosis of mullerian adenocarcinoma was made. The patient died from recurrent disease 9 months after surgery. Conclusion.: Regardless of primary focus, adenosarcoma with sarcomatous overgrowth is associated with postoperative recurrence and a fatal outcome. [Copyright &y& Elsevier]

Objective: To report a series of four patients with uterine sarcoma, including one with müllerian adenosarcoma (MA) and three with low-grade endometrial stromal sarcoma (LGESS), who developed intracranial meningiomas while receiving the progesterone agonist megestrol acetate. Methods: The hospital records, imaging studies, and pathology slides of four patients who were treated for uterine sarcomas and subsequently developed intracranial meningiomas were reviewed. Results: All patients underwent surgery for their gynecologic cancers and received maintenance therapy with long-term hormonal suppression with megestrol acetate. Each of the four patients later developed neurologic symptoms secondary to intracranial meningiomas. Three patients had more than one meningioma. Histopathologic examination of all excised tumors showed strong immunoreactivity for progesterone receptors (PRs). Conclusions: Patients with uterine sarcoma subtypes LGESS and MA may be predisposed to develop meningiomas, particularly in the setting of long-term treatment with megestrol acetate. Alternatively, preexisting, clinically silent meningiomas in these patients may have progressed to the point of clinical symptoms in the presence of the progesterone agonist megestrol acetate. Without previous imaging studies showing the presence or absence of meningioma before initiation of megestrol acetate treatment, there is no way to draw definitive conclusions regarding this possibility. Clinical and neuroradiologic surveillance for meningiomas should be strongly considered in patients with these uterine sarcoma subtypes, particularly in patients undergoing long-term suppressive therapy with megestrol acetate. [Copyright &y& Elsevier]